Subclinical synovial proliferation in patients with severe haemophilia A: The value of ultrasound screening and biochemical markers

Author:

van Bergen Eline D. P.1ORCID,van Leeuwen Flora H. P.2ORCID,Foppen Wouter2ORCID,Timmer Merel A.1ORCID,Schutgens Roger E. G.1ORCID,Mastbergen Simon C.3,Lafeber Floris P. J. G.3,de Jong Pim A.2,Fischer Kathelijn1ORCID,van Vulpen Lize F. D.1ORCID

Affiliation:

1. Center for Benign Haematology, Thrombosis and Haemostasis, Van Creveldkliniek University Medical Center Utrecht University Utrecht Utrecht The Netherlands

2. Department of Radiology & Nuclear Imaging University Medical Center Utrecht University Utrecht Utrecht The Netherlands

3. Department of Rheumatology & Clinical Immunology University Medical Center Utrecht University Utrecht Utrecht The Netherlands

Abstract

AbstractAimSubclinical bleeding and inflammation play a role in progression of haemophilic arthropathy. Synovial proliferation is predictive of joint bleeding and its early detection may guide treatment changes and prevent arthropathy progression. This study evaluated the prevalence of active and inactive subclinical synovial proliferation and investigated potential biochemical blood/urine markers to identify patients with active subclinical synovial proliferation.MethodsThis cross‐sectional study included patients with severe haemophilia A born 1970–2006 who were evaluated during routine clinic visits. Patients with (a history of) inhibitors or recent joint bleeding were excluded. Elbows, knees and ankles were examined for subclinical synovial proliferation by ultrasound and physical examination. Active synovial proliferation was distinguished from inactive synovial proliferation using predefined criteria. Blood/urine biochemical markers (serum osteopontin, sVCAM‐1, Coll2‐1, COMP, CS846, TIMP, and urinary CTX‐II) were compared individually and as combined indexes between patients with and without active synovial proliferation.ResultsThis cohort consisted of 79 patients with a median age of 31 years (range 16.5–50.8 years) with 62/79 (78%) of the patients using continuous prophylaxis. The annualized joint bleeding rate over the last 5 years was .6 (.2–1.1). Active (17/79, 22%) and inactive subclinical synovial proliferation (17/79, 22%) were both prevalent in this cohort. Biochemical markers were not correlated with active subclinical synovial proliferation.ConclusionSubclinical synovial proliferation, both active and inactive, was prevalent in patients with severe haemophilia A with access to prophylaxis and would be overlooked without routinely performed ultrasounds. Biochemical markers were unable to identify patients with active subclinical synovial proliferation.

Publisher

Wiley

Subject

Genetics (clinical),Hematology,General Medicine

Reference37 articles.

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4. Clotting factor concentrates given to prevent bleeding and bleeding‐related complications in people with hemophilia A or B;Iorio A;Cochrane Database Syst Rev,2011

5. Prophylaxis versus Episodic Treatment to Prevent Joint Disease in Boys with Severe Hemophilia

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